{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,20]],"date-time":"2026-09-20T16:39:36Z","timestamp":1789922376479,"version":"4.0.1"},"reference-count":49,"publisher":"The Company of Biologists","issue":"14","license":[{"start":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T00:00:00Z","timestamp":1784592000000},"content-version":"vor","delay-in-days":6,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000780","name":"European Union","doi-asserted-by":"crossref","award":["861329"],"award-info":[{"award-number":["861329"]}],"id":[{"id":"10.13039\/501100000780","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100003245","name":"Ministerie van Onderwijs, Cultuur en Wetenschap","doi-asserted-by":"crossref","award":["024.006.034 Lifelong VISION"],"award-info":[{"award-number":["024.006.034 Lifelong VISION"]}],"id":[{"id":"10.13039\/501100003245","id-type":"DOI","asserted-by":"crossref"}]},{"name":"ZonMW","award":["04630042210010"],"award-info":[{"award-number":["04630042210010"]}]},{"DOI":"10.13039\/501100001832","name":"Radboud University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001832","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,7,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Mutations in the ciliary gene INPP5E, encoding inositol polyphosphate-5-phosphatase E (INPP5E), can cause retinal degeneration as part of the ciliopathy Joubert syndrome or non-syndromic retinitis pigmentosa (RP). INPP5E regulates the membrane makeup of the primary cilium; however, its function in the specialized sensory photoreceptor cells of the human retina remain unclear. Here, we utilize control and CRISPR\/Cas9-generated INPP5E knockout (INPP5ED477N\/D477N) human induced pluripotent stem cells (iPSCs) to generate retinal organoids (ROs). Through proteomic and immunofluorescence analysis, we show that INPP5E plays an important role in early retinal development and photoreceptor progenitor cell differentiation. In mature ROs, INPP5E localizes to the connecting cilium of photoreceptors, and the loss of INPP5E leads to altered localization of ARL13B and rhodopsin in mature photoreceptors. Furthermore, photoreceptor outer segment structure is affected, leading to elongated outer segment membranes in both cone and rod photoreceptors, suggesting an important role for INPP5E in photoreceptor outer segment membrane biogenesis. Together, these data underline the importance of INPP5E in retina development and photoreceptor structure and highlight the usability of ROs to study protein function in a human context.<\/jats:p>","DOI":"10.1242\/jcs.264431","type":"journal-article","created":{"date-parts":[[2026,6,30]],"date-time":"2026-06-30T08:37:38Z","timestamp":1782808658000},"source":"Crossref","is-referenced-by-count":2,"title":["Loss of INPP5E affects photoreceptor outer segment membrane biogenesis in iPSC-derived human retinal organoids"],"prefix":"10.1242","volume":"139","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2100-6597","authenticated-orcid":false,"given":"Kae R.","family":"Whiting","sequence":"first","affiliation":[{"name":"Research Institute for Medical Innovation, Radboud University Medical Center 1 Department of Human Genetics , , Nijmegen 6525 GA ,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5675-2031","authenticated-orcid":false,"given":"Mariam G.","family":"Aslanyan","sequence":"additional","affiliation":[{"name":"Research Institute for Medical Innovation, Radboud University Medical Center 1 Department of Human Genetics , , Nijmegen 6525 GA ,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lynn","family":"van Summeren","sequence":"additional","affiliation":[{"name":"Research Institute for Medical Innovation, Radboud University Medical Center 1 Department of Human Genetics , , Nijmegen 6525 GA ,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6302-8997","authenticated-orcid":false,"given":"Tina","family":"Beyer","sequence":"additional","affiliation":[{"name":"Eberhard Karls University of T\u00fcbingen, Institute for Ophthalmic Research 2 , T\u00fcbingen 72074 ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9681-2908","authenticated-orcid":false,"given":"Katrin","family":"Dahlke","sequence":"additional","affiliation":[{"name":"Eberhard Karls University of T\u00fcbingen, Institute for Ophthalmic Research 2 , T\u00fcbingen 72074 ,","place":["Germany"]},{"name":"Eberhard Karls University of T\u00fcbingen, Core Facility for Medical Proteomics 3 , T\u00fcbingen 72074 ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6590-8309","authenticated-orcid":false,"given":"Thomas","family":"Theil","sequence":"additional","affiliation":[{"name":"Institute for Neuroscience and Cardiovascular Sciences, University of Edinburgh 4 , Hugh Robson Building, Edinburgh EH8 9XD ,","place":["UK"]},{"name":"Simons Initiative for the Developing Brain, University of Edinburgh 5 , Hugh Robson Building, Edinburgh EH8 9XD ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2693-689X","authenticated-orcid":false,"given":"Karsten","family":"Boldt","sequence":"additional","affiliation":[{"name":"Eberhard Karls University of T\u00fcbingen, Institute for Ophthalmic Research 2 , T\u00fcbingen 72074 ,","place":["Germany"]},{"name":"Eberhard Karls University of T\u00fcbingen, Core Facility for Medical Proteomics 3 , T\u00fcbingen 72074 ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5178-8163","authenticated-orcid":false,"given":"Ronald","family":"Roepman","sequence":"additional","affiliation":[{"name":"Research Institute for Medical Innovation, Radboud University Medical Center 1 Department of Human Genetics , , Nijmegen 6525 GA ,","place":["The Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2026,7,21]]},"reference":[{"key":"2026072105584718100_JCS264431C1","doi-asserted-by":"publisher","first-page":"iyad031","DOI":"10.1093\/genetics\/iyad031","article-title":"The gene ontology knowledgebase in 2023","volume":"224","author":"Aleksander","year":"2023","journal-title":"Genetics"},{"key":"2026072105584718100_JCS264431C2","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1038\/75556","article-title":"Gene ontology: tool for the unification of biology","volume":"25","author":"Ashburner","year":"2000","journal-title":"Nat. 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